Intellectual Disability-related genes increase ADHD risk and locomotor activity in <i>Drosophila</i>

Klein, Marieke; Singgih, Euginia L.; Rens, Anne van; Demontis, Ditte; Børglum, Anders D.; Mota, Nina Roth; Castells-Nobau, Anna; Kiemeney, Lambertus A.; Brunner, Han G.; Arias-Vásquez, Alejandro; Schenck, Annette; Voet, Monique van der; Franke, Barbara

doi:10.1101/725937

Cited by 1 publication

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“…This pleads for an impact of a targeted, specific remodelling of niche architecture during development on adult behaviour. Hyperactivity (increased locomotor activity and reduced sleep) in Drosophila has been found in diverse models of neuro/developmental and neurological disorders, including the Fragile X syndrome [62], Attention-deficit/hyperactivity disorder (ADHD) [63] and Shwachman-Diamond syndrome [64]. In all these cases, changes in locomotion were correlated with synaptic and axonal abnormalities.…”

Section: Discussionmentioning

confidence: 99%

A corset of adhesions during development establishes individual neural stem cell niches and controls adult behaviour

Banach-Latapy

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Guénal

et al. 2022

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Neural stem cells (NSCs) reside in a defined cellular microenvironment, the niche, which supports the generation and integration of neuronal lineages. The mechanisms building a sophisticated niche structure around NSCs, and their functional relevance for neurogenesis are yet to be understood. In the Drosophila larval brain, the cortex glia (CG) encase individual NSC lineages, organizing the stem cell population and newborn neurons into a stereotypic structure. We first found that lineage information is dominant over stem cell fate. We then discovered that, in addition to timing, the balance between multiple adhesion complexes supports the individual encasing of NSC lineages. An intra-lineage adhesion through homophilic Neuroglian interactions provides strong binding between cells of a same lineage, while a weaker interaction through Neurexin-IV exists between CG to NSC lineages. Their loss leads to random, aberrant grouping of several NSC lineages together, and to altered axonal projection of newborn neurons. Further, we link the loss of these two adhesion complexes during development to locomotor hyperactivity in the resulting adults. Altogether, our findings identify a corset of adhesions building a neurogenic niche at the scale of individual stem cell and provide the proof-of-principle that mechanisms supporting niche formation during development define adult behaviour.

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Section: Discussionmentioning

confidence: 99%

A corset of adhesions during development establishes individual neural stem cell niches and controls adult behaviour

Banach-Latapy

Rincheval

Guénal

et al. 2022

Preprint

View full text Add to dashboard Cite

show abstract

Intellectual Disability-related genes increase ADHD risk and locomotor activity in Drosophila

Abstract: Disclosures and acknowledgmentsAcknowledgements Funding: Part of this work was carried out on the Dutch national e-infrastructure with the support of SURF Foundation. The Nijmegen Biomedical Study is a population-based survey conducted at the

Cited by 1 publication

References 47 publications

A corset of adhesions during development establishes individual neural stem cell niches and controls adult behaviour

A corset of adhesions during development establishes individual neural stem cell niches and controls adult behaviour

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